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Pediatr Surg Int (2005) 21: 400402 DOI 10.

1007/s00383-004-1339-8

CASE REPORT

F. A. Abantanga

Ileal invagination of the sigmoid colon producing a sigmoidorectal intussusception combined with rectal prolapse in a 3-year-old child

Accepted: 21 February 2003 / Published online: 21 December 2004 Springer-Verlag 2004

Abstract Intussusception is very common in children all over the world, especially those under 2 years. This can be ileoileal, ileocolic, or colocolic, i.e. the immediate proximal part of the intestine telescoping into the distal portion of the intestine. Ileosigmoid intussusception involves the invagination of a loop of the ileum into the sigmoid colon, producing two obstructions: that involving the invaginated loop of terminal ileum and that of the sigmoidorectal intussusception. This can lead to ischaemia and necrosis of either the ileum or sigmoid colon. The case presented here is suspected to be the rst one reported in the world scientic literature. Keywords Invagination Sigmoidorectal intussusception Rectal prolapse Ileosigmoid knotting

Case report
A 3-year-old boy, who is the 5th of six siblings, was admitted to the casualty recovery ward of Komfo Anokye Teaching Hospital with a provisional diagnosis of rectal prolapse. The history revealed that the child had been passing stool one morning when a mass suddenly protruded through the anus. This protruding mass had grown bigger since its appearance, and medical advice was sought only on the 3rd day after failure of traditional treatment. The protruding mass was associated with neither abdominal pain nor vomiting. This was the rst and only episode of rectal prolapse in this child. Examination revealed a moderately malnourished, ill-looking child with no signs of distress at all. The abdomen was not distended, was soft all over, and was nontender. An umbilical hernia was present. There was a bluish oedematous mass about 15 cm in length protruding from the anus (Fig. 1). Rectal examination revealed no sulcus between the prolapsed mass and the

Introduction
Ileal invagination of the sigmoid colon leading to sigmoidorectal intussusception is a rare disease entity unlike ileosigmoid knotting, which is relatively common in certain African, Asian, and Middle Eastern countries [14]. A search of the world literature (including a Medline search) revealed no cases in children of sigmoidorectal intussusception in combination with an ileal invagination into the sigmoid colon together with a rectal prolapse. A case of such a pathology in a 3-year-old boy from Kumasi, Ghana, is presented.

F. A. Abantanga Department of Surgery, School of Medical Sciences, Kwame Nkrumah University of Science & Technology, Kumasi, Ghana E-mail: [email protected]

Fig. 1 Intestine protruding from the anal orice on admission to hospital

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Fig. 2 The proposed mechanism of ileal invagination of the sigmoid colon resulting in a sigmoidorectal intussusception

rectal canal; that is, a blockage prevented the examining nger from going very high up. This conrmed the diagnosis of a rectal prolapse. Taking into consideration the fact that the mass had been present for the past 3 days and that it was oedematous and blue, the child was quickly resuscitated and prepared for possible reduction of the mass under general anaesthesia. Under general anaesthesia, a repeat rectal examination conrmed the earlier ndings, but an attempt at reducing the mass back into the abdominal cavity failed. This attempt was abandoned, and the peritoneal cavity was entered through a low abdominal midline incision. It was discovered that a loop of the terminal ileum, about 20 cm long, had invaginated the sigmoid colon at the rectosigmoid junction, thus causing an intussusception of the sigmoid into the rectum (Fig. 2). The intussusceptum progressed further, taking along with it the terminal ileum and the rectum, hence the ndings at rectal examination. With combined gentle traction on the loop of ileum and pressure on the mass in the perineum, it was successfully reduced but with some diculty (Fig. 3). The ileum was assessed to be normal, though oedematous and ischaemic; the distal portion of the sigmoid colon and the proximal part of the rectum were found to be necrotic. The necrotic portion of bowel was resected; the proximal part of the rectum, just below the peritoneal reection, was closed and left in the pelvic cavity. An end sigmoid colostomy was also constructed. Histopathological examination of the resected portion of the bowel revealed no pathology. Twelve weeks after

Fig. 3 Components of the unusual intussusception

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the initial surgery and with improvement in the childs general condition, intestinal continuity was restored. The child has been reviewed several times after discharge from hospital and is doing well, although he is still small for his age.

Discussion
As stated above, there are no known cases in the literature of ileal invagination of the sigmoid colon combined with a sigmoidorectal intussusception and rectal prolapse in a child. The patient reported here is likely to be the rst case. This is a case of intussusception involving both the small and large bowel, but not the typical ileocaecal/ileocolic intussusception, which is considered common among children [5]. Isolated large bowel intussusceptions are also known to be common among children. A series published by Davies and Cywes [6] reported an unusually high incidence of sigmoid intussusception in infants in Africa. Sigmoidorectal intussusception is the least common type of intussusception that occurs in infants and children [7]. In both of these types of intussusception (sigmoid and sigmoidorectal), there is usually a delay in diagnosis because of inadequate assessment of the prolapsed mass [6] or lack of clinicians awareness of the existence of this type of intussusception [7]. In the present case, contributory factors to this type of pathology may have been a very mobile terminal ileum with an elongated mesentery and a long redundant sigmoid colon, which is known to be a common and normal nding among black Africans.

There are no features specic for this condition, and the diagnosis was only established at laparotomy. This case was rst diagnosed as a rectal prolapse because of the digital rectal examination ndings of an absent sulcus between the prolapsed bowel and the wall of the anal canal. The absence of the classical triad of intussusceptioncolicky abdominal pains, a palpable mass, and bleeding per rectumalso compounded the issue. The presence of a high index of suspicion and consideration of the length of the prolapsed bowel and the curved nature of the prolapse, as seen in Fig. 1, will denitely aid in making the diagnosis of intussusception preoperatively. All in all, aggressive resuscitation and prompt surgery should be carried out to ensure a good prognosis in such cases.

References
1. Gibney EJ, Mock CN (1993) Ileosigmoid knotting. Dis Colon Rectum 36:855857 2. NGuessan HA, Keli E, Yapo P, et al. (1992) Ileosigmoid knot. Chirurgie 118:382388 3. Alver A, Oren D, Tireli M, et al. (1993) Ileosigmoid knotting in Turkey. Review of 68 cases. Dis Colon Rectum 36:11391147 4. Puthu D, Rajan N, Shenou GM, Pai SU (1991) The ileosigmoid knot. Dis Colon Rectum 34:161166 5. Grant HW, Buccimazza I, Hadley GP (1996) A comparison of colo-colic and ileo-colic intussusception. J Pediatr Surg 31:1607 1610 6. Davies MR, Cywes S (1978) Colonic intussusception in children. S Afr Med J 54:517519 7. Singh RB, et al. (1995) Sigmoidorectal intussusception. Ind J Gastroent 14:75

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